Development of a core outcome set for congenital pulmonary airway malformations: study protocol of an international Delphi survey

Introduction A worldwide lack of consensus exists on the optimal management of asymptomatic congenital pulmonary airway malformation (CPAM) even though the incidence is increasing. Either a surgical resection is performed or a wait-and-see policy is employed, depending on the treating physician. Man...

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Główni autorzy: René M H Wijnen, Maarten Schurink, Nagarajan Muthialu, Pietro Bagolan, Nazan Cobanoglu, Dhanya Mullassery, Holger Till, M. Singh, M. Boon, Sergei Hermelijn, Casper Kersten, Silvia Gartner, Carmen Mesas Burgos, Alberto Sgro, Stijn Heyman, Janne Suominen, Liesbeth Desender, Paul Losty, Kjetil Ertresvag, Harm A W M Tiddens, Marco Schnater, S.M. Hermelijn, C.M. Kersten, J.M. Schnater, R.M.H. Wijnen, H.A.W.M. Tiddens, S.C.M. Cochius, J. Suominen, M. Pakarinen, L. Martelius, S. Heyman, D. Vervloessem, H. Steyaert, A. Sgrò, P. Gamba, M. Schurink, S. van der Heide, J. Roukema, N. Rikkers-Mutsaerts, S. Terheggen-Lagro, S. de Beer, E. Haarman, H. Till, G. Singer, M. Metzelder, P Sezen, L. Desender, H. Schaballie, N. Cobanoglu, G. Gollu, M. Stanton, A. Bonnard, R. Sfeir, N. Muthialu, D. Mullassery, C. Wallis, D. Cox, P. Bagolan, F. Morini, C. Mesas Burgos, P. Conner, E. Caffrey Osvald, C. Bitkover, H. Decaluwé, M. Proesmans, J. Deprest, S. Gartner, A. Lain, P.D. Losty, I. Sinha, I. Yardley, L. Wessel, K. Zahn, T. Schaible, N. Qvist, M. Zampoli, G. Aksnes, C. K. van der Ent, K.M. Winter-de Groot, R. Peters, E. Hannon, Q. Jöbsis, M. Bannier
Format: Artykuł
Język:English
Wydane: BMJ Publishing Group 2021-04-01
Seria:BMJ Open
Dostęp online:https://bmjopen.bmj.com/content/11/4/e044544.full
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author René M H Wijnen
Maarten Schurink
Nagarajan Muthialu
Pietro Bagolan
Nazan Cobanoglu
Dhanya Mullassery
Holger Till
M. Singh
M. Boon
Sergei Hermelijn
Casper Kersten
Silvia Gartner
Carmen Mesas Burgos
Alberto Sgro
Stijn Heyman
Janne Suominen
Liesbeth Desender
Paul Losty
Kjetil Ertresvag
Harm A W M Tiddens
Marco Schnater
S.M. Hermelijn
C.M. Kersten
J.M. Schnater
R.M.H. Wijnen
H.A.W.M. Tiddens
S.C.M. Cochius
J. Suominen
M. Pakarinen
L. Martelius
S. Heyman
D. Vervloessem
H. Steyaert
A. Sgrò
P. Gamba
M. Schurink
S. van der Heide
J. Roukema
N. Rikkers-Mutsaerts
S. Terheggen-Lagro
S. de Beer
E. Haarman
H. Till
G. Singer
M. Metzelder
P Sezen
L. Desender
H. Schaballie
N. Cobanoglu
G. Gollu
M. Stanton
A. Bonnard
R. Sfeir
N. Muthialu
D. Mullassery
C. Wallis
D. Cox
P. Bagolan
F. Morini
C. Mesas Burgos
P. Conner
E. Caffrey Osvald
C. Bitkover
H. Decaluwé
M. Proesmans
J. Deprest
S. Gartner
A. Lain
P.D. Losty
I. Sinha
I. Yardley
L. Wessel
K. Zahn
T. Schaible
N. Qvist
M. Zampoli
G. Aksnes
C. K. van der Ent
K.M. Winter-de Groot
R. Peters
E. Hannon
Q. Jöbsis
M. Bannier
author_facet René M H Wijnen
Maarten Schurink
Nagarajan Muthialu
Pietro Bagolan
Nazan Cobanoglu
Dhanya Mullassery
Holger Till
M. Singh
M. Boon
Sergei Hermelijn
Casper Kersten
Silvia Gartner
Carmen Mesas Burgos
Alberto Sgro
Stijn Heyman
Janne Suominen
Liesbeth Desender
Paul Losty
Kjetil Ertresvag
Harm A W M Tiddens
Marco Schnater
S.M. Hermelijn
C.M. Kersten
J.M. Schnater
R.M.H. Wijnen
H.A.W.M. Tiddens
S.C.M. Cochius
J. Suominen
M. Pakarinen
L. Martelius
S. Heyman
D. Vervloessem
H. Steyaert
A. Sgrò
P. Gamba
M. Schurink
S. van der Heide
J. Roukema
N. Rikkers-Mutsaerts
S. Terheggen-Lagro
S. de Beer
E. Haarman
H. Till
G. Singer
M. Metzelder
P Sezen
L. Desender
H. Schaballie
N. Cobanoglu
G. Gollu
M. Stanton
A. Bonnard
R. Sfeir
N. Muthialu
D. Mullassery
C. Wallis
D. Cox
P. Bagolan
F. Morini
C. Mesas Burgos
P. Conner
E. Caffrey Osvald
C. Bitkover
H. Decaluwé
M. Proesmans
J. Deprest
S. Gartner
A. Lain
P.D. Losty
I. Sinha
I. Yardley
L. Wessel
K. Zahn
T. Schaible
N. Qvist
M. Zampoli
G. Aksnes
C. K. van der Ent
K.M. Winter-de Groot
R. Peters
E. Hannon
Q. Jöbsis
M. Bannier
collection DOAJ
description Introduction A worldwide lack of consensus exists on the optimal management of asymptomatic congenital pulmonary airway malformation (CPAM) even though the incidence is increasing. Either a surgical resection is performed or a wait-and-see policy is employed, depending on the treating physician. Management is largely based on expert opinion and scientific evidence is scarce. Wide variations in outcome measures are seen between studies making comparison difficult thus highlighting the lack of universal consensus in outcome measures as well. We aim to define a core outcome set which will include the most important core outcome parameters for paediatric patients with an asymptomatic CPAM.Methods and analysis This study will include a critical appraisal of the current literature followed by a three-stage Delphi process with two stakeholder groups. One surgical group including paediatric as well as thoracic surgeons, and a non-surgeon group including paediatric pulmonologists, intensive care and neonatal specialists. All participants will score outcome parameters according to their level of importance and the most important parameters will be determined by consensus.Ethics and dissemination Electronic informed consent will be obtained from all participants. Ethical approval is not required. After the core outcome set has been defined, we intend to design an international randomised controlled trial: the COllaborative Neonatal NEtwork for the first CPAM Trial, which will be aimed at determining the optimal management of patients with asymptomatic CPAM.
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spelling doaj.art-06c9a7aad0b64236b2f683b76fbd709b2024-11-18T08:40:07ZengBMJ Publishing GroupBMJ Open2044-60552021-04-0111410.1136/bmjopen-2020-044544Development of a core outcome set for congenital pulmonary airway malformations: study protocol of an international Delphi survey 0René M H Wijnen1Maarten Schurink2Nagarajan Muthialu3Pietro Bagolan4Nazan Cobanoglu5Dhanya Mullassery6Holger Till7M. Singh8M. BoonSergei Hermelijn9Casper Kersten10Silvia Gartner11Carmen Mesas Burgos12Alberto Sgro13Stijn Heyman14Janne Suominen15Liesbeth Desender16Paul Losty17Kjetil Ertresvag18Harm A W M Tiddens19Marco Schnater20S.M. HermelijnC.M. KerstenJ.M. SchnaterR.M.H. WijnenH.A.W.M. TiddensS.C.M. CochiusJ. SuominenM. PakarinenL. MarteliusS. HeymanD. VervloessemH. SteyaertA. SgròP. GambaM. SchurinkS. van der HeideJ. RoukemaN. Rikkers-MutsaertsS. Terheggen-LagroS. de BeerE. HaarmanH. TillG. SingerM. MetzelderP SezenL. DesenderH. SchaballieN. CobanogluG. GolluM. StantonA. BonnardR. SfeirN. MuthialuD. MullasseryC. WallisD. Cox21P. BagolanF. MoriniC. Mesas BurgosP. ConnerE. Caffrey OsvaldC. BitkoverH. DecaluwéM. ProesmansJ. DeprestS. GartnerA. LainP.D. LostyI. SinhaI. YardleyL. WesselK. ZahnT. SchaibleN. QvistM. ZampoliG. AksnesC. K. van der EntK.M. Winter-de GrootR. PetersE. HannonQ. JöbsisM. Bannier3University College LondonDepartment of Paediatric Surgery and Intensive Care, Erasmus MC University Medical Center, Rotterdam, The NetherlandsPediatric Surgery, Radboud University Medical Centre Amalia Childrens Hospital, Nijmegen, The NetherlandsGreat Ormond Street Hospital for Children NHS Foundation Trust, UKDepartment of Medical and Surgical Neonatology, Ospedale Pediatrico Bambino Gesu, Roma, ItalyPediatric Pulmonology, Ankara University Faculty of Medicine, Ankara, TurkeyPediatric Surgery, Great Ormond Street Hospital for Children, London, UKPediatric Surgery, Medical University of Graz, Graz, AustriaThe Garvan Institute of Medical Research, Immunogenomics, Sydney, AustraliaPediatric Surgery, Erasmus MC Sophia Childrens Hospital, Rotterdam, The NetherlandsPediatric Surgery, Erasmus MC Sophia Childrens Hospital, Rotterdam, The NetherlandsPediatric Pulmonology, Hospital Universitari Vall dHebron, Barcelona, SpainPediatric Surgery, Karolinska Institutet, Stockholm, SwedenPediatric Surgery, Padua University Hospital, Padova, ItalyPediatric Surgery, ZNA-GZA Paola Childrens Hospital, Antwerp, BelgiumPediatric Surgery, University of Helsinki Childrens Hospital, Helsinki, FinlandPediatric Surgery, Ghent University Faculty of Medicine and Health Sciences, Gent, BelgiumDepartment of Paediatric Surgery, Institute Of Systems Molecular and Integrative Biology, University of Liverpool, Liverpool, UKPediatric Surgery, Oslo University Hospital, Oslo, NorwayPediatric Pulmonology, Erasmus MC Sophia Childrens Hospital, Rotterdam, The NetherlandsPediatric Surgery, Erasmus MC Sophia Childrens Hospital, Rotterdam, The NetherlandsPfizer Inc, Collegeville, United States of AmericaIntroduction A worldwide lack of consensus exists on the optimal management of asymptomatic congenital pulmonary airway malformation (CPAM) even though the incidence is increasing. Either a surgical resection is performed or a wait-and-see policy is employed, depending on the treating physician. Management is largely based on expert opinion and scientific evidence is scarce. Wide variations in outcome measures are seen between studies making comparison difficult thus highlighting the lack of universal consensus in outcome measures as well. We aim to define a core outcome set which will include the most important core outcome parameters for paediatric patients with an asymptomatic CPAM.Methods and analysis This study will include a critical appraisal of the current literature followed by a three-stage Delphi process with two stakeholder groups. One surgical group including paediatric as well as thoracic surgeons, and a non-surgeon group including paediatric pulmonologists, intensive care and neonatal specialists. All participants will score outcome parameters according to their level of importance and the most important parameters will be determined by consensus.Ethics and dissemination Electronic informed consent will be obtained from all participants. Ethical approval is not required. After the core outcome set has been defined, we intend to design an international randomised controlled trial: the COllaborative Neonatal NEtwork for the first CPAM Trial, which will be aimed at determining the optimal management of patients with asymptomatic CPAM.https://bmjopen.bmj.com/content/11/4/e044544.full
spellingShingle René M H Wijnen
Maarten Schurink
Nagarajan Muthialu
Pietro Bagolan
Nazan Cobanoglu
Dhanya Mullassery
Holger Till
M. Singh
M. Boon
Sergei Hermelijn
Casper Kersten
Silvia Gartner
Carmen Mesas Burgos
Alberto Sgro
Stijn Heyman
Janne Suominen
Liesbeth Desender
Paul Losty
Kjetil Ertresvag
Harm A W M Tiddens
Marco Schnater
S.M. Hermelijn
C.M. Kersten
J.M. Schnater
R.M.H. Wijnen
H.A.W.M. Tiddens
S.C.M. Cochius
J. Suominen
M. Pakarinen
L. Martelius
S. Heyman
D. Vervloessem
H. Steyaert
A. Sgrò
P. Gamba
M. Schurink
S. van der Heide
J. Roukema
N. Rikkers-Mutsaerts
S. Terheggen-Lagro
S. de Beer
E. Haarman
H. Till
G. Singer
M. Metzelder
P Sezen
L. Desender
H. Schaballie
N. Cobanoglu
G. Gollu
M. Stanton
A. Bonnard
R. Sfeir
N. Muthialu
D. Mullassery
C. Wallis
D. Cox
P. Bagolan
F. Morini
C. Mesas Burgos
P. Conner
E. Caffrey Osvald
C. Bitkover
H. Decaluwé
M. Proesmans
J. Deprest
S. Gartner
A. Lain
P.D. Losty
I. Sinha
I. Yardley
L. Wessel
K. Zahn
T. Schaible
N. Qvist
M. Zampoli
G. Aksnes
C. K. van der Ent
K.M. Winter-de Groot
R. Peters
E. Hannon
Q. Jöbsis
M. Bannier
Development of a core outcome set for congenital pulmonary airway malformations: study protocol of an international Delphi survey
BMJ Open
title Development of a core outcome set for congenital pulmonary airway malformations: study protocol of an international Delphi survey
title_full Development of a core outcome set for congenital pulmonary airway malformations: study protocol of an international Delphi survey
title_fullStr Development of a core outcome set for congenital pulmonary airway malformations: study protocol of an international Delphi survey
title_full_unstemmed Development of a core outcome set for congenital pulmonary airway malformations: study protocol of an international Delphi survey
title_short Development of a core outcome set for congenital pulmonary airway malformations: study protocol of an international Delphi survey
title_sort development of a core outcome set for congenital pulmonary airway malformations study protocol of an international delphi survey
url https://bmjopen.bmj.com/content/11/4/e044544.full
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